Urologists evaluate men with various sexual dysfunctions, including complaints of inability to obtain and/or maintain a satisfactory erection during sexual stimulation. There is, however, a subset of men who have the opposite problem. Such men have a condition of unwanted, unremitting sensations of erection in the absence of sexual desire, thoughts or fantasies. These men may be on the verge of orgasm or experience spontaneous orgasm, often with limited to no resolution and aggravation of symptoms by sexual activity. Symptoms are often provoked by specific circumstances, such as sitting, driving, listening to music, general anxiety, stress or nervousness. This condition is unlike priapism where an unwanted, often painful prolonged erection is observed on physical examination. This condition, termed persistent genital arousal disorder (PGAD)/genito-pelvic dysesthesia (GPD), reveals inconsistent evidence of erection on physical examination. PGAD was first described in women in 2001 but PGAD has been commonly noted in men as well. A recent study assessed the prevalence of PGAD criteria in 2 large, nonclinical samples of Canadian undergraduate students (1,634, Study 1) and a nationally representative sample from the U.S. (1,026, Study 2). It was found that 1.1%–4.3% of men and 0.6%–2.7% of women reported PGAD. This prevalence translates to multiple millions of people who potentially experience PGAD/GPD. Furthermore, PGAD/GPD is often associated with significant, negative psychosocial impact involving despair, emotional lability, catastrophization and/or suicidal ideation. For all these reasons it is important to review the contemporary urological management of men with complaints of PGAD/GPD.

This review examines the case of a male patient with PGAD/GPD who underwent diagnosis and treatment in our sexual medicine facility, with over 3-year treatment followup. The patient, a 38-year-old single male in a monogamous relationship for 10 years, presented to his urologist in 2018 with a 5-year history of feelings of spontaneous genital arousal 24/7, especially in the evening and during the night, making sleep difficult. His internist had treated him with gabapentin, sertraline, alprazolam and amitriptyline with some reduction of dysesthesia symptoms. He constantly felt like he was approaching an orgasm but did not have spontaneous ejaculation. He had symptoms of bladder urgency and frequency. He further complained of urethral discomfort described as increased arousal in the urethra triggered by ejaculation. In addition, he described sharp shooting, burning pains in the penis, perineum, anus, and behind the right and left thigh and calf. Many evenings he experienced a nearly irresistible urge to move his legs, consistent with restless leg syndrome. Of note, he could not spread his toes. The patient noted that his symptoms were made worse with anxiety, vibration from a phone or hair clippers, touching of his nipples, bowel movements and sitting on a hard chair. He had excellent morning erections and 100% penile rigidity, sustaining ability and spontaneity for sexual activity. Total testosterone was 388 ng/dl (range 250–1,100 ng/dl). His lumbar MRI showed a high-intensity zone consistent with an annular tear in the L4-5 epidural space (Fig. 1).

Figure 1. The high-intensity zone is a characteristic finding of an annular tear representing the presence of nucleus pulposus material in the epidural space.

The contemporary pathophysiology of PGAD is explained by a common underlying neurological basis. Using functional MRI in patients with PGAD, spontaneous, intense and extensive activation of the paracentral lobule in the somatosensory cortex of the brain was identified, even in the absence of any overt physical genital stimulation. In contrast, functional MRI in patients without PGAD revealed only minimal activation of the paracentral lobule when imagining just genital stimulation. The goal of PGAD management is to identify and then treat the triggers that are causing this intense spontaneous activation of the paracentral lobule. These triggers originate in 1 or more of 5 regions in each PGAD/GPD patient: 1) end organ, 2) pelvis/perineum, 3) cauda equina, 4) spinal cord and/or 5) brain. (Fig. 2)

Figure 2. Triggers of PGAD/GPD are generated from 1 or more of 5 regions. The trigger in this case history emanates from region 3. AVM, arterio-venous malformation. SSRI, selective serotonin reuptake inhibitor. SNRI, serotonin and norepinephrine reuptake inhibitor.

This PGAD/GPD patient described dysesthesia within the sensory fields of the somatic pudendal nerve (ie penis, perineum, anus), visceral afferent pelvic nerve (ie bladder, urethra) and somatic sciatic nerve (ie right and left thigh and calf). Of note, he experienced issues in the motor field of the sciatic nerve (ie inability to spread the toes and restless leg syndrome). The pudendal, pelvic and sciatic nerves merge at the S2-3 foramina in the cauda equina (region 3), where they coalesce to form the S2-3 sacral nerve roots. These sacral spinal nerve roots ascend to the first synapse in the conus medullaris and, along the way, may be subject to compression or impingement radiculopathies from cauda equina pathologies, such as his L4-5 annular tear (Fig. 1). Thus, based on the patient’s symptoms involving the sensory fields of the pudendal, pelvic, and sciatic nerves and his lumbar MRI, a lumbosacral annular tear-induced sacral radiculopathy was suspected as the primary trigger for his PGAD/GPD.

The urologist can perform minimally-invasive testing to better understand the triggers of PGAD/GPD. Having a multidisciplinary team involving a psychologist, spine surgeon and sexual medicine physician/urologist would facilitate biopsychosocial management. When the patient has both penile and urethral dysesthesia symptoms the urologist can perform both a local penile and urethral anesthesia test with lidocaine. Should the patient experience clinically significant symptom reduction, this would suggest that the trigger for the PGAD/GPD was in region 1. In this case, the patient continued to have dysesthesia symptoms after the penile and urethral anesthesia tests, consistent with “upstream” pathology. The urologist can either perform or refer the patient for assessment of the integrity of the pudendal and sciatic nerves via minimally invasive neuro-genital testing (quantitative sensory testing, sacral dermatome testing and bulbocavernosus reflex latency testing). These neurogenital tests are fully described in Goldstein et al, 2021.¹ Finally, a pain medicine specialist could perform a diagnostic transforaminal epidural spinal injection of 1 ml of lidocaine under fluoroscopic control into the epidural space at L4-5. This patient experienced 70% resolution of symptoms for 4 hours, consistent with the diagnosis of lumbosacral annular tear-induced sacral radiculopathy. The patient eventually underwent lumbar endoscopic spine surgery and was discharged the same day. He had no postoperative complications. He had immediate reduction of his PGAD/GPD symptoms with marked resolution of his anxiety, bother and distress. After 3 years his patient global impression of improvement score remains “2” (much better).

In summary, PGAD/GPD can be an extremely distressing sexual health condition for men. Urologists should understand the contemporary pathophysiology and utilize a consensus-based algorithm involving a multidisciplinary team for management of men with this condition.